Effectiveness of constraint-induced movement therapy compared with bimanual intensive training in children with spastic unilateral cerebral palsy - a systematic review

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!

Department of Health and Care Sciences

Effectiveness of constraint-induced movement therapy compared with bimanual intensive training in children with spastic unilateral cerebral palsy - a systematic review

Maria Tervahauta

Master thesis in Health Sciences

Clinical Neurological Physiotherapy in relation to children May 2014

(word count: 21 338)

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“The wonder of being lies in aliveness and the wonder of aliveness originates in movement.”

- Maxine Sheets-Johnstone, 1999

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Acknowledgements

First and foremost, I offer my sincerest gratitude to my supervisor Mr. Gyrd Thrane, who has supported me throughout my work on this thesis with his knowledge and expertise. While he has constantly directed me to the right direction with his insightful, critical comments and advice, he has allowed me the room to find my own way of working, and to make my mistakes. This thesis would not have materialised without his constant encouragement and effort. I could not have wished for a better supervisor for this project.

I am grateful for Mrs. Robbin Hickman who has been my contact person for the American Academy for Cerebral Palsy and Developmental Medicine methodology, and who has tiredly answered my questions especially regarding the quality of conduct questions.

A big thank you to Mr. Torstein Låg, the principle librarian at the Psychology and Law library at the University of Tromsø, for providing me with extremely helpful advice about literature searching, and for reviewing the search strategies for the diﬀerent databases. Thank you for ensuring me that the search strings need not look super-complex and ﬁll pages after pages.

I would like to give special thanks also to Inger Lise Rorgemoen, a specialist in paediatric physiotherapy, who has been my supervisor in the practical studies as part of this master’s degree. I have gained invaluable insights into paediatric physiotherapy through our many discussions and reﬂections about physiotherapy practice and the role of paediatric physiotherapists. These discussions have helped me tremendously in the thought process for this thesis.

I would like to especially thank Pascal Bolla for being the second reviewer for the quality of conduct ratings and for giving his time to help even though he has been busy with working on his own master’s degree.

Lastly, a big thanks to my family and friends for keeping me sane during this project, for taking me on trips to the mountains, by the sea and in the forest, and for supplying me with wine, weissbier, and generally good conversations that mostly had nothing to do with physiotherapy. It was often during these times that I found new inspirations for this project.

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Abstract

AIM The aim of this study was to systematically assess the eﬀectiveness of Constraint-Induced

Movement Therapy (CIMT) compared with Bimanual Intensive Training (BIT) across the domains of the International Classiﬁcation of Functioning, Disability and Health - Children and Youth (ICF-CY) in children with spastic unilateral cerebral palsy (CP).

METHOD A systematic literature review using the American Academy for Cerebral Palsy and Developmental Medicine (AACPDM) methodology was conducted. The following databases were searched: CINAHL, Cochrane Library, EMBASE, MEDLINE, PEDro, Pubmed and PsycINFO. The primary outcomes of interest were bimanual upper extremity (UE) function, unimanual UE function and participation. The secondary outcomes of interest were individualised goals and quality of life. The quality of conduct of the included trials was rated by two independent reviewers using the AACPDM rating questions. The standardised mean diﬀerence (SMD) and 95% conﬁdence intervals (95%CI) were calculated using post-scores.

RESULTS Six RCTs met the inclusion criteria, one with high, three with moderate and two with low quality of conduct. The dose of the intervention varied from a total of 60 hours over 10 days to 210 hours over 10 weeks. Follow-up varied from immediate post-intervention to 52 weeks. No adverse events were reported. The measurement instruments used assessed items across the ICF-CY domains making it not possible to comprehensively report the findings using the ICF-CY. In general, no or minimal differences in the effectiveness of CIMT compared with BIT were observed in the outcomes of interest at any follow-up. The 95%CIs were consistently wide. The mean group pre-post change scores exceeded a known minimal detectible change (MDC) in both groups only when attainment of

individualised scores was measured with the COPM. It was not possible to calculate SMD for the quality of life outcomes. A meta-analysis was assessed to be little relevant due to high variability in dosing, the used measurement instruments and the variability in the children’s response.

INTERPRETATION Based on the findings in the RCTs included in this systematic review, it is not possible to confidently say that CIMT or BIT is more effective than the other in improving manual ability, participation and individual goals in children with unilateral CP at group-level.

KEYWORDS unilateral cerebral palsy, constraint-induced movement therapy, bimanual intensive training, upper extremity function, ICF

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Sammendrag

FORMÅL Formålet med denne studien var å utforske eﬀekten av Constraint-Induced Movement Therapy (CIMT) sammenlignet med Bimanual Intensiv Trening (BIT) på tvers av kategoriene i den Internasjonale Klassiﬁkasjonen av Funksjon, Funksjonshemming og Helse – for barn og ungdom (ICF- CY) hos barn med unilateral cerebral parese (CP).

METODE En systematisk litteraturgjennomgang ble gjennomført ved bruk av American Academy for Cerebral Palsy and Developmental Medicine (AACPDM) metodologi. Det ble søkt i følgende databaser:

CINAHL, Cochrane Library, EMBASE, MEDLINE, PEDro, Pubmed og PsycINFO. Eﬀekt av intervensjonene på bimanuell hånd- og armfunksjon, unimanuell hånd- og armfunksjon og deltagelse var av størst interesse. Individuelle mål og livskvalitet var av sekundær interesse. Kvaliteten på de inkluderte studiene ble vurdert av to uavhengige personer ved bruk av AACPDM ‘quality of conduct’ spørsmål.

Standardised mean diﬀerence (SMD) og 95% konﬁdensintervall (95%CI) ble beregnet av post-skårer.

RESULTATER Seks RCT-studier ble inkludert, én med høy, tre med moderat og to med lav kvalitet.

Intervensjonsmengden varierte fra 60 timer fordelt på 10 dager til 210 timer fordelt på 10 uker.

Oppfølgingsperioden varierte fra umiddelbart etter avsluttet intervensjon opp til 52 uker etter. Det ble ikke rapportert om negative bivirkninger. Målingsinstrumentene som ble benyttet vurderte elementer på tvers av kategoriene i ICF-CY, noe som gjorde det umulig å rapportere resultatene i tilknytting til de spesiﬁkke ICF-CY kategoriene. Generelt, ingen eller minimal forskjell mellom CIMT sammenlignet med BIT ble observert. 95% kondifensintervall av SMD var konstant brede. Gjennomsnittlig pre-post endring i begge gruppene overskred minste målbare endring kun når individuelle mål ble vurdert med COPM.

Det var ikke mulig å beregne SMD for instrumentene som målte livskvalitet. En meta-analyse ble vurdert til å være av lite relevans på grunn av store variasjoner i intervensjonsmengden,

målingsinstrumentene som ble brukt og barnas respons til intervensjonene.

FORTOLKNING Basert på resultater fra RCTer som ble funnet i denne systematiske

litteraturgjennomgangen, er det ikke mulig å si at CIMT eller BIT har bedre eﬀekt enn den andre for å bedre hånd- og armfunksjon, deltagelse og oppnåelse av individuelle mål hos barn med unilateral CP på gruppenivå.

NØKKELORD unilateral cerebral palsy, constraint-induced movement therapy, bimanual intensive training, upper extremity function, ICF

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Preface

This thesis was prepared in accordance with the guidelines for Master thesis by UiT - the Arctic University of Norway. Based on the guidelines the thesis is an individual piece of scientiﬁc work of a self-chosen theme up to 25 000 words (excluding the front page, acknowledgements, table of contents, bibliography and supplementary material). In line with the UiT guidelines, all references follow the APA format.

The chapters of this thesis are organised as follows:

Chapter 1 provides an overview of the topic with a general introduction.

Chapter 2 provides the theoretical background upon which the thesis is build.

Chapter 3 explains the methods used.

Chapter 4 presents the results.

Chapter 5 provides a discussion of the ﬁndings.

Chapter 6 provides a short conclusion for the ﬁndings.

The following supplementary material is provided:

I. Search strategy for each database II. References for qualitative studies

III. List of publications from the research groups IV. Reasoning for quality of conduct scoring

All references are compiled in a bibliography at the end of the thesis.

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Supplementary material I - Search strategy for each database 60 Supplementary material II - References to for qualitative studies 63 Supplementary material III - Excluded publications from the research groups 64 Supplementary material IV - Reasoning for scoring of the quality of conduct questions 65

Bibliography 73

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Abbreviations

AACPDM American Academy of Cerebral Palsy and Developmental Medicine AHA Assisting Hand Assessment

Assessment of Life Habits for Children BIT Bimanual Intensive Training

CI conﬁdence interval

CIMT Constrained-Induced Movement Therapy CNS Central Nervous System

CEBM Centre for Evidence Based Medicine CP Cerebral Palsy

COPM Canadian Occupational Performance Measure EBM Evidence-Based Medicine

EBP Evidence-Based Practice

GMFCS E&R Gross Motor Function Classiﬁcation Scale Extended and Revised ICF International Classiﬁcation of Functioning, Disability and Health

ICF-CY Classiﬁcation of Functioning, Disability and Health - Children and Youth The Jebsen–Taylor Test of Hand Function

MACS Manual Ability Classiﬁcation System MCID Minimal clinically important diﬀerence

MUUL Melbourne Assessment of Unilateral Upper Limb function PEDI Pediatric Evaluation of Disability Inventory

PRISMA Preferred Reporting Items for Systematic reviews and Meta-Analyses statement QoL Quality of life

QUEST Quality of Upper Extremity Skills Test RCT Randomised controlled trial

SCPE Surveillance of Cerebral Palsy in Europe SD Standard deviation

SDC smallest detectable change SMD Standardised mean diﬀerence UE Upper extremity

WHO World Health Organization

WCPT World Confederation of Physical Therapy

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1. Introduction

Moving, touching and manipulating objects is fundamental to our ability to participate in and learn about the world (Sheets-Johnstone, 1999, pp. 226-229). We engage in daily activities, occupations and social contact with the aim to do something useful for ourselves and for others. The full meaning of manual competency, therefore, is created in the context of our relationships with others. Furthermore, doing something useful is the key to building, shaping and expressing identity. Developing identity though daily activities and occupations thus creates the context for a meaningful life, and as Christiansen (1999, p. 547) discusses further, life meaning helps us to be well.

Most children with spastic unilateral cerebral palsy (CP) have the intellectual capacity to attend regular schools, and essentially the same societal demands and expectations are placed on these children as on non-disabled children (Sakzewski, Ziviani, & Boyd, 2009, p.

e1111; Skold, Josephsson, & Eliasson, 2004, p. 416). Whereas the majority of the children with unilateral CP achieve independent walking, impaired upper extremity (UE) function

contributes to about half of the children experiencing limitations in activities of daily living and restrictions in participation in education, leisure and occupational activities (Fedrizzi, Pagliano, Andreucci, & Oleari, 2003, p. 85; Gordon, 2010, p. 315; Pagliano et al., 2001;

Sakzewski et al., 2009, p. e1111).

Because of the non-progressive disturbances in the foetal or the developing infant brain, children with unilateral CP experience intrinsic limitations in their manual ability, such as impairments in muscle tone, strength, sensation, motor planning and control as well as coordination of UE movements (Gordon, 2011, p.57; Sakzewski et al., 2009, p. e1111;

Steenbergen, Verrel, & Gordon, 2007). Above unimanual tasks, these impairments typically contribute to limitations in activities where the use of the two hands is required (Facchin et al., 2009, p. 217; Gordon, 2010, p. 315; Steenbergen, Charles, & Gordon, 2008).

Together with the impairments, behavioural aspects constitute an important factor aﬀecting manual ability in children with unilateral CP. With a vast majority of activities in daily living,

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education and occupations comprising of bimanual tasks, a mismatch between the children’s capabilities and the task and societal demands may arise. In their interviews with children with unilateral CP, Skold et al. (2004, p. 423) describe that some children choose to omit difficult activities because they wish to conceal their disability and inability to complete activities akin to non-disabled children. As the children repeatedly experience difficulties in using the affected UE, many choose to find other solutions and/or adjust their behaviour to meet the task demands (Fedrizzi, Rosa-Rizzotto, Turconi, Pagliano, Fazzi, Pozza, Facchin, et al., 2013, p. 171; Houwink, Aarts, Geurts, & Steenbergen, 2011, p. 2158; Skold et al., 2004; Taub, Uswatte, Mark, & Morris, 2006). With constant negative reinforcement many children

disregard the aﬀected UE, even when the aﬀected UE is only mildly impaired. This

phenomenon is described as developmental disregard (Hoare, Wasiak, Imms, & Carey, 2007, p. 676; Houwink et al., 2011, p.2158), comparable to ‘learned non-use’ in adults after

cerebrovascular accident (Taub et al., 2006; Taub, Uswatte, & Pidikiti, 1999, p. 239).

Importantly, the lack of spontaneous use of the aﬀected UE leads to asymmetrical development of the upper extremities, and may further contribute to secondary musculoskeletal impairments.

Throughout much of the 20th century motor impairments, especially in the UE, were thought to be static, with little potential for improvement (Gordon & Magill, 2012, p. 161). However, increasing evidence now demonstrate that manual ability is a dynamic phenomenon and may be changed by practice (Eliasson, 2007). Importantly, improvements in hand function have been observed to occur over a longer period of time than previously expected Eliasson et al.

(2006, p. e1234). Based on the current motor learning theories, improving manual ability requires intensive training of targeted activities where children are provided with adequate opportunity, motivation, experience, and environment (Hoare et al. 2007; Sakzewski, Ziviani, &

Boyd, 2014).

In the last decade, constraint-induced movement therapy (CIMT) and bimanual intensive training (BIT) have been proposed as suitable interventions. CIMT was developed from behavioural learning theories and discoveries in neuroscience, and is said to represent “a paradigm shift” in UE rehabilitation (Brady & Garcia, 2009). The focus is in reversing the

behavioural aspects associated with the limited UE use by constraining, and thereby inducing,

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increased use of the aﬀected UE. Based on the fact that most daily activities are bimanual tasks, BIT was developed with the focus to improve the use of the aﬀected UE as a support hand in functional bimanual activities (Charles & Gordon, 2006; Gordon, Schneider, Chinnan,

& Charles, 2007). In line with the current motor learning theories, both interventions are based on intensive, repetitive, goal-oriented task practice where the children are

incrementally provided with more challenging tasks.

Both CIMT and BIT have been reported to be more eﬀective in improving UE function in children with unilateral CP than no treatment or very basic treatment (Hoare et al., 2007;

Sakzewski et al., 2014). Although the results are promising, it remains unclear whether the eﬃcacy of CIMT is due to the constraint or the intensive training (Gordon, 2011; Ferdizzi et al., 2013, p. 162; Sakzewski et al., 2009, p. e1118).

Recent primary research has therefore focused on comparing CIMT with BIT. To gather the results from randomised controlled trials (RCTs), Dong et al. (2013) conducted a systematic review with a narrative analysis. Although narrative analyses provide potentially valuable information, they do not allow for assessing the consistency of the intervention effects across the primary studies. Sakzewski et al. (2014) included RCTs comparing CIMT and BIT as part of their large meta-analysis. As the aim of the meta-analysis was to gathered data on all non- invasive UE interventions for children with unilateral CP, it did not include a comprehensive analysis of the RCTs comparing CIMT and BIT. To date, no systematic review has based its findings on the International Classification of Functioning, Disability and Health - Children and Youth (ICF-CY) (WHO, 2007). The ICF-CY provides an invaluable framework for standardised description of health and functioning, as well as a unified language for communication of research findings.

To fill the research gap, a systematic review was conducted with the aim to examine the effectiveness of constraint-induced movement therapy (CIMT) compared with bimanual intensive training (BIT) in children with spastic unilateral CP assessed on the different ICF-CY domains. The Guideline for Developing Systematic Reviews of Treatment Interventions for Children with Developmental Disabilities by the American Academy of Cerebral Palsy and Developmental Medicine (AACPDM) Treatment Outcome Committee (Darrah, Hickman,

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O'Donnell, Vogtle, & Wiart, 2008) was used as the basis in this systematic review. The aim of the guideline is to produce systematic reviews with focus on using the ICF-CY as a framework and language for reporting ﬁndings in paediatric research.

Based on previous literature and theory of training specificity, it was hypothesised that CIMT and BIT show similar efficiency in improving UE function in children with unilateral CP, but that CIMT is more effective in improving unimanual UE function and BIT in improving bimanual UE function.

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2. Theory

All observation is selectively orientated and no theory-free data exists, therefore, all research and interpretation of the evidence, is essentially directed by the researcher’s though models (Thornquist, 2003, p. 197). This chapter provides an overview of the theoretical framework used in this thesis, as well as of other theory the work was build upon. The framework was build upon a variety of current theory in health care and rehabilitation sciences.

2.1. The International Classiﬁcation of Functioning, Disability and Health

The International Classiﬁcation of Functioning, Disability and Health (ICF) (WHO, 2001)

provides the main conceptual framework and language for this thesis. The decision followed recommendations by the World Confederation of Physical Therapy (WCPT) that supported the implementation of the ICF in 2003 (Escorpizo et al., 2010, p. 1054). More speciﬁcally, the terminology of the version adapted for children and youth, the ICF-Children and Youth (ICF- CY), published by the WHO in 2007, was applied.

The ICF is build on the perspective that human functioning and disability consist of

multidimensional, complex, and dynamic interactions between diﬀerent domains of health that interact with environmental and personal factors (ﬁgure 1). Body functions comprise of physiological functions, e.g. cognitive functions, and body structures constitute of anatomical parts, e.g. organs or limbs. Activities entail execution of a task or action by an individual, and participation implies involvement in a life situation. Personal factors describe the background of an individual’s life and living, and comprise of features of the individual that are not part of a health condition or health state. Environmental factors comprise of the physical, social and attitudinal environments in which people live. The contextual factors (personal and

environmental factors) may either facilitate or be barriers to functioning (WHO, 2001;

Rosenbaum & Stewart, 2004, p. 9). The domains are divided into chapters that are further organised into hierarchical categories which include increasingly detailed deﬁnitions on second, third, and sometimes fourth level.

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Figure 1. The ICF-CY model displaying the interactions between the domains. Adapted from WHO (2007).

The ICF emphasises the context in which functioning and disability occur. The concept of performance is used to describe what an individual does in his or her current, habitual environment, including the societal context. This includes involvement in life situations and the lived experience of the person in the context of his or her actual environment. In contrast, the concept of capacity describes the person’s ability to function in a standardised

environment (WHO, 2001, p. 11). Knowledge about the person’s capacity and his or her

performance enables determination of any gaps between the person’s best abilities and what he or she actually does in everyday life.

It is now well-recognised that a common language is essential in advancing the science of disablement, in communication between therapists and others working with the clients, and in the documentation of the health care processes (Jette, 2006, 2009; Rosenbaum & Stewart, 2004; WHO, 2001). A common language is essential for a uniform understanding of the results in primary studies as well as for assisting clinicians in the interpretation of the results in the context of other available resources. The ICF provides a framework for such a standard

language (Wagner & Davids, 2012, pp. 1257-1258). However, for an eﬃcient use of the ICF, it is essential that the users completely understand what the perspective of functioning of the ICF entails and what is does not entail. For instance, the ICF regards functioning as a distinct concept from quality of life and health preferences in the future. Functioning in the ICF

perspective is related to the limitations and restrictions related to a health problem, where as

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quality of life refers to how someone feels about these limitations and restrictions (Cieza et al., 2005, p. 212).

Over the last decades a range of, often competing, condition-specific and generic instruments have been developed to measure changes in functioning. With such a large selection of measurement instruments available, it has become increasingly difficult for researchers to select the most appropriate instruments for their trials, and for readers to interpret and compare the results of different trials (Cieza et al., 2005, p. 212). The “ICF linking rules” (Cieza et al., 2005) were therefore developed. The aim of the linking rules is to facilitate linking of outcome measures to the ICF domains. Importantly, as the measurement instruments used should reflect the aims of the interventions, the linking rules provide “a connecting framework between interventions and outcome measures, facilitating the selection of the most

appropriate outcome measure for the aim of the intervention” (Cieza et al., 2005, p. 213).

2.2. Cerebral palsy

Deﬁnition

The deﬁnition of cerebral palsy (CP) was updated in 2005 to adjust to the advancements in the knowledge of physiological and pathological brain development, as well as to adapt to the changing concepts about impairments, functional status and participation brought upon by the ICF (Rosenbaum et al., 2007). Thus, in this thesis CP is recognised as “a permanent disorder of movement and posture causing activity limitations attributed to non-progressive disturbances having occurred in the developing foetal or infant brain. The motor disorders of cerebral palsy are often accompanied by disturbances of sensation, perception, cognition, communication, and behaviour; by epilepsy; and by secondary musculoskeletal problems” (Bax et al., 2005;

Rosenbaum et al. 2007, p. 9).

Prevalence

CP is one of the most common causes of childhood physical disability (Oskoui, Coutinho, Dykeman, Jette, & Pringsheim, 2013, p. 509; SCPE, 2000, p. 816). A methodologically rigorous systematic review and meta-analysis based on population-based registries found an overall prevalence of 2.11 per 1000 live births (95% CI 1.98–2.25) (Oskoui et al., 2013, p. 511). A

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population-based study in Norway found a prevalence of 2.1 per 1000 live births (Andersen et al., 2008, p. 6). Despite signiﬁcant advances in pre- and perinatal care and falls in neonatal mortality generally, the prevalence of CP has remained stable over the past 40 years (Sigurdardottir, Thorkelsson, Halldorsdottir, Thorarensen, & Vik, 2009, p. 356).

Approximately 50% of the children with CP are born at term (Sigurdardottir et al., 2009, p.

357) with the prevalence of CP being highest among children born before 28 weeks of

gestation (111.80 per 1000 live births; 95% CI 69.53–179.78), and lowest among children born after 36 weeks of gestation (1.35 per 1000 live births; 95% CI 1.15–1.59). In terms of birth weight, the prevalence is highest in children weighing 1000 to 1499g (59.18 per 1000 live births; 95% CI 43.38–73.95), and lowest in children weighing over 2500g (1.33 per 1000 live births; 95% CI 1.19–1.49) (Oskoui et al., 2013, p. 511).

Although the overall prevalence of CP has remained stable, changes in the characteristics of CP have occurred. The proportion of children with diplegia has increased, possibly reflecting increased survival of pre-term and very pre-term infants. The prevalence of children with quadriplegia has decreased, presumably due to advances in health care (Sigurdardottir et al., 2009, p. 356; Wright & Wallmann, 2012, p. 581). A population-based study in Norway studied the characteristics of CP in the Norwegian population and found that of all children with CP 33% (95% CI: 28–39) were classified with spastic unilateral CP, 49% (95% CI: 41–53) with spastic bilateral CP, 6% (95% CI: 4–10) with dyskinetic CP and 5% (95% CI: 3–8) with ataxic CP. The subtype could not be classified in 7% (95% CI: 4–11) of the children (Andersen et al., 2008, pp.

6-7). A similar prevalence of spastic unilateral CP has been reported in other countries (Hoare et al., 2007, p. 675; Sakzewski et al., 2009, p. e1111).

Etiology

The causes of CP are not completely understood. However, associations have been made to certain events arising prenatally in the foetus, perinatally at around birth, postnatally after birth or postneonatally more than 28 days after birth (Aarli, Andersen, Jansen, & Sommerfelt, 2010, p. 227; Wright & Wallmann, 2012, p. 578). Brain damage occurring after the child is two years old is classiﬁed as acquired brain injury (Aarli et al., 2010, p. 227). Some of the most important etiological factors include hypoxia or disturbance of circulation to the brain,

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developmental disturbance in the brain without a genetic cause, infection or reaction to an infection, genetic causes, toxic causes (e.g. medication of the mother during pregnancy), and traumatic causes occurring during the ﬁrst two years of life (Aarli et al., 2010, p. 227). The developmental stage of the brain at the time of the cerebral insult is decisive for the

evolvement and prognosis of the characteristics of the child’s cerebral palsy (Aarli et al., 2010, p. 227; Feys et al., 2010, p. 176).

Classiﬁcation

The Surveillance of Cerebral Palsy in Europe (SCPE) (SCPE, 2000) classiﬁcation of subtypes of CP, the Gross Motor Function Classiﬁcation Scale Extended and Revised (GMFCS E&R)

(Palisano, Rosenbaum, Bartlett, & Livingston, 2008) and the Manual Ability Classification Scale (MACS) (Eliasson, Forssberg, et al., 2006) were used as the main systems to communicate classification of children with CP in this thesis. Considering that gross motor function and manual ability do not fully correlate in children with CP (Carnahan, Arner, & Hagglund, 2007), together the classification systems provide a complementary communication system for the clinic and for research and administration purposes (Rosenbaum, Palisano, Bartlett, Galuppi,

& Russell, 2008, p. 249).

The Surveillance of Cerebral Palsy in Europe (SCPE). The SCPE classiﬁcation system on subtypes of CP was developed by a European-wide collaboration group in 2000 (SCPE, 2000).

The four subtypes of CP are deﬁned as follows (Cans et al., 2007, pp. 36-37):

- Spastic CP is characterised by increased tone and pathological reflexes, observed as increased reflexes, e.g. hyperreflexia, or pyramidal signs, e.g. positive Babinski. Depending on the involvement of the limbs, the terms spastic unilateral or bilateral CP are used.

- Ataxic CP is characterised by loss of orderly muscular coordination so that movements are performed with abnormal force, rhythm and accuracy.

- Dyskinetic CP is characterised by involuntary, uncontrolled, recurring, and occasionally stereotyped movements. The primitive reﬂex patterns predominate and the muscle tone is varying. Dyskinetic CP is further divided into dystonic CP dominated by abnormal postures and hypertonia, and choreo-athetotic CP dominated by hyperkinesia and hypotonia.

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The Gross Motor Function Classiﬁcation Scale Extended and Revised (GMFCS E&R). The GMFCS E&R is a valid, reliable and clinically meaningful system to classify gross motor

function in children with CP aged 2 to 18 years (Palisano et al., 2008; Rosenbaum et al., 2008, p. 250). Focus is on the child’s usual performance in sitting, walking and wheeled mobility and on self-initiated movement. The system is divided into ﬁve levels with each level being

described speciﬁcally for four diﬀerent age groups to account for the fact that although CP is a non-progressive condition, motor function changes over time (Rosenbaum et al., 2008, p.

250). A full description of the GMFCS E&R is available from www.canchild.ca. The general headings for the GMFCS levels are described as follows:

- Level I: the child walks without limitations.

- Level II: the child walks with limitations.

- Level III: the child walks with a hand-held mobility device.

- Level IV: the child has limited ability for independent mobility, and may require an electrical wheelchair.

- Level V: the child has limited ability to maintain antigravity head and trunk postures, and are transported in manual wheelchair in all settings.

The Manual Ability Classiﬁcation Scale (MACS). The MACS is a valid, reliable and clinically meaningful system used to classify manual ability in children with CP aged 4 to 18 years of age. The focus is on assessing the child’s self-initiated ability to handle objects in daily

activities, and the need for assistance or adaptation in performing bimanual manual activities in everyday life (Eliasson, Krumlinde-Sundholm, et al. 2006; Ohrvall, Krumlinde-Sundholm, &

Eliasson, 2013). The function of the two hands is not separated but considered together. The MACS is limited to situations where objects are manipulated within the reach of the child, this to ensure minimal influence of gross motor function on the classification of fine motor skills (Eliasson et al., 2006, p. 553). A complete description of the MACS is available from

www.macs.nu. The MACS levels are generally described as follows:

- Level I: the child handles objects easily and successfully.

- Level II: the child handles most objects but with somewhat reduced quality and/or speed.

- Level III: the child handles objects with diﬃculty, and needs assistance to prepare and/or modify activities.

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- Level IV: the child handles a limited selection of easily managed objects in adapted situations.

- Level V: the child does not handle objects and has severely limited ability to perform even simple actions.

Other classiﬁcation systems. In addition to the MACS, other systems have been used to classify UE function in children with unilateral CP. The House classiﬁcation (House, Gwathmey,

& Fidler, 1981) is used to evaluate the function in the affected hand after surgery for thumb- in-palm deformity. The Zancolli classification (Zancolli & Zancolli, 1981) is used to grade the level of muscle tone in the wrist and finger flexors as well as active wrist and finger extension.

These systems classify aspects of grasping instead of functional performance, and have not been tested for reliability (Eliasson, Krumlinde-Sundholm, et al., 2006, p. 549). A detailed description of the House and the Zancolli classiﬁcations is provided for example in Arner et al.

(2008, p. 1340).

The classiﬁcation systems & the ICF-CY

When considering these classification systems on the ICF-CY, it is clear that the SCPE classification is based on the body functions domain, while the GMFCS E&R and the MACS describe functioning primarily on the activities and participation domain. Both GMFCS E&R and MACS focus on assessing the child’s performance in his or her habitual environment, distinct from the child’s capacity (the best ability assessed in a standardised setting). Although the classification systems focus on activities and participation, some of the concepts used to distinguish between the GMFCS and MACS levels belong to the ICF-CY body functions or environmental factors domain. For instance, the distinction between GMFCS levels II, III and IV in children aged 6 to 12 years is based on the child’s need for mobility devices, a concept belonging to the environmental factors domain. The distinction between the MACS and GMFCS levels I and II in children aged 6 to 12 years is based on the quality of movement including concepts such as speed, balance and coordination. Of these, coordination and balance belong to the body functions domain (b755 Involuntary movement reaction functions, b760 Control of voluntary movement functions, b765 Involuntary movement functions). Speed of movement is not currently classified on the ICF.

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2.3. Manual ability

Adapting the conceptual framework employed by Eliasson et al. (2006) in the MACS, the deﬁnition of manual ability by Penta et al. (2001) is used in this thesis. Penta et al. (2001, p.

1627) deﬁned manual ability as “the ability to manage daily activities that require the use of the upper limbs, whatever the strategies involved”. The quality and extend of manual ability depends on a complex interplay between several factors within the individual, the task and the environment (Shumway-Cook & Woollacott, 2012, pp. 477-478). Because manual ability comprises a behaviour, it should be observed, and assessed, as a person’s performance of activities in his or her everyday context (Eliasson et al., 2006, p. 549; Penta et al., 2001, p.

1627).

Knowledge of the development of manual ability is imperative for understanding the

possibilities and possible eﬀects of UE interventions. While genetics provide the child with a general blueprint for neural development, the development of the unique neural patterns in the brain is largely driven by experience and environmental requirements (Galea, 2014, p. 71).

The brain is a dynamic, self-organising system that spontaneously adapts and changes to the requirements it is presented with in pre- and postnatal life (Braun & Bock, 2011, pp. 14-15).

All development is mediated by ongoing, bi-directional interchange between hereditary factors and the environment (Braun & Bock, 2011, p. 14). Edelman’s theory of neuronal group selection (Edelman, 1989) highlights the interaction between nature and nurture in the

development of any skills. First, the ‘primary repertoires’ of neuronal groups are developed as epigenetic factors regulate the structural diversity of the neural groups via cell division, cell diﬀerentiation, programmed cell death and neuronal migration. Second, in the post-natal life the strength of the developed synaptic connections in the neuronal groups are modiﬁed based on the requirements they are predisposed to, forming the ‘secondary repertoires’ of neuronal groups. Third, interconnected series of neuronal groups develop dynamic neural

“maps” that independently receive inputs from the real world.

Development of fine motor skills in typically developing children is characterised by rapid progression during the first years of life followed by refinement of skills throughout the childhood (Eliasson, Forssberg, et al., 2006, p. e1227; Holmefur, Krumlinde-Sundholm,

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Bergstrom, & Eliasson, 2010, p. 352). Some elements of manual ability, particularly locating objects in space and transport of the arm, may be present at rudimentary form at birth with other movement components, such as grasp, develop during the ﬁrst year of life (Shumway- Cook & Woollacott, 2012, p. 505). As described by Boyd et al. (2014) UE skills in general develop in several non-consecutive stages including 1) discovering the hand; 2) visually regarding the hand; 3) visually exploring objects in space; 4) swiping at objects; 5) contacting objects; 6) ineﬀectively grasping objects; and 7) developing prehensile movements to better grasp objects.

Relatively little is known about the specific development of manual ability in children with unilateral CP (Eliasson, Forssberg, et al., 2006, p. e1227), and few tools exist for prediction purposes (Holmefur et al., 2013, p. 72). Holmefur et al. (2010) performed a longitudinal study in which they observed that children who used the affected UE for grasping at 18 months of age, had a more rapid development and reached a higher level of manual ability than children who did not use their affected hand. Children who had a higher manual ability reached 90%

of their ability limit at 3 years of age, whereas children who had a lower manual ability

developed more slowly, reaching 90% of their ability limit at 7 years of age. Importantly, it has been observed that improvements in manual ability continue even after adolescence

regardless of the initial severity of the child’s hand function (Eliasson, Forssberg, et al., 2006, p. e1232). The subsequent decline or improvement of skills after this time may be dependent on both severity of impairment and access to ongoing intervention (Eliasson, Forssberg, et al., 2006, p. e1234; Fedrizzi, Pagliano, Andreucci, & Oleari, 2003, p. 91).

A longitudinal study compared data from neuroimaging with the children’s scores on the Assisting Hand Assessment (AHA) and found associations between the type, location and size of the perinatal brain lesion and the development of manual ability (Holmefur et al., 2013, p.

76). Children with white-matter lesions tend to obtain higher manual ability limits than children with middle cerebral artery (MCA) lesions (Feys et al., 2010; Holmefur et al., 2013, p.

76; Holmstrom et al., 2010). However, independent of the type of the brain lesion, the strongest predictor for development of better manual ability was the absence of combined involvement of the basal ganglia and the thalamus (Feys et al., 2010; Holmefur et al., 2013, pp.

75-76).

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Considering that the thalamus is central for providing the cerebral cortex with the key information of representations of body space and extra-personal space, both of which essential for all complex behaviour (Galea, 2014, p. 73), this structural impairment

corresponds with the ﬁnding that children with unilateral CP commonly present with deﬁcits in movement planning (Steenbergen & Gordon, 2006, p. 780). As Steenbergen & Gordon (2006, p. 781) describe, movement planning involves anticipation of the future state of the motor system, and the consequences of an action, and is an essential for any function because of the inherent delays in the sensorimotor system. With bimanual tasks being generally more complex than unimanual tasks and requiring higher levels of motor planning, it is not surprising that children with unilateral CP typically have limitations especially in activities where the use of the two hands is required (Gordon, 2010, p. 315; Facchin et al., 2009, p. 217; Steenbergen et al., 2008).

Experience of actions is essential for development of internal models used to anticipate the consequences of those actions. Even though many children with unilateral CP present with a mild impairment of the more aﬀected UE, many choose not to use the UE to its full potential in daily life, a phenomenon described as developmental disregard (Hoare et al., 2007, p. 676;

Houwink et al., 2011, p. 2158). With the missing experience of actions, deficits in the development of internal models may add to further impairments in motor planning and manual ability. Importantly, research has shown that children with unilateral CP may have the capacity to improve their manual ability with sufficient practice. For example, in a study by Duff & Gordon (2003) a group of children with unilateral CP were not initially able to

appropriately plan their fingertip forces on their affected UE to pick up different objects, yet they were able to do so with sufficient practice.

2.4. Assessing manual ability

The complexity of manual ability together with the heterogeneous population of children with unilateral CP makes assessment of manual ability a challenge (Vargus-Adams, 2009). A wide range of instruments are available to measure diﬀerent aspects of manual ability and function in this population, yet the constructs and psychometric properties of these

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instruments vary widely. A range of measurement instruments are presented in the methods section in this thesis and recent systematic reviews provide additional information, see for example James et al. (2013), Lemmens (2012), Carlon et al. (2010), Gilmore et al. (2010), Klingels et al. (2010), Baird et al. (2010), Davis et al. (2010), Harvey et al. (2008), and Sakzewski et al. (2007).

To obtain accurate information, the measurement instruments must be valid in that they measure what they are meant to measure, and reliable in that it can be ensured that the measurements are stable in time and between diﬀerent persons completing the

measurements. The instruments should be easily and readily employable and be sensitive to detecting change in function. The smallest detectable change (SDC) is an estimate of the smallest amount of change that can be considered a real change in function, and not a measurement error. The minimal clinically important diﬀerence (MCID) deﬁnes a threshold

“that says when a person/group has just begun to experience what is an important improvement” (Beaton, Boers, & Wells, 2002, p. 109; Vargus-Adams, 2009, p. 7).

2.5. Evidence-based practice

In the past 20 years evidence-based practice (EBP) has become an essential ingredient of all health care practice (Dijkers, Murphy, & Krellman, 2012), and the evolving paradigm is the driving force of many physiotherapy vision statements, for instance in the American Physical Therapy Association’s (APTA) Vision 2020 (APTA, 2014). The international description of physical therapy, as part of the policy statement of the World Confederation for Physical Therapy (WCPT), advocates that all member organisations’ definition of physiotherapy must emphasise the need for practice to be evidence-based whenever possible (WCPT, 2013). In their much-cited definition, the original Evidence-based Medicine Working group at McMaster University in Canada defined evidence-based medicine as

“the conscientious, explicit, and judicious use of current best evidence in making decisions about the care and individual patients. The practice of evidence based medicine means integrating individual clinical expertise

with the best available external clinical evidence from systematic research” (Sackett, Rosenberg, Gray, Haynes, & Richardson, 1996, p. 71).

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Different professions have adjusted the definition during the years, yet all definitions are variations of the original. Importantly, most rehabilitation professions have integrated

language concerning patient values in their deﬁnitions of EBP (Dijkers et al., 2012, p. S165). In the original article, Sackett et al. (1996, p. 72) write

“...without clinical expertise, practice risks becoming tyrannised by evidence, for even excellent external evidence may be inapplicable to or inappropriate for an individual patient”.

Thus, to ensure full appreciation of the concept of EBP, it is essential to emphasise that EBP implies integration of findings attainted through systematic research, knowledge attained through professional practice and expertise, and the values of the individual patient (figure 2). Using his or her clinical reasoning skills and clinical expertise to consider these three aspects, the physiotherapist considers, and re-considers, whether the findings from research may be applicable for the individual patient (Howick et al., 2011; Jewell, 2011, pp. 26-29).

Figure 2. Components of evidence-based practice.

As Dijkers et al. (2012, p. S166) point out, statements of what constitute ‘evidence’ differ depending of the time, place and culture. Traditionally, the model of “best evidence” has been fixed, and a categorical interpretation of evidence, in that studies using a certain type of research method give a claim more evidential support, has been a driving force in EBP (Kerry, Eriksen, Lie, Mumford, & Anjum, 2012, p. 1007). However, with EBP having expanded from simple pharmaceutic treatments to areas of health care dealing with complex interventions, it is well-recognised that different types of methods are needed to produce evidence for

diﬀerent types of clinical questions (Dijkers et al., 2012, p. S168; Hart & Bagiella, 2012). As emphasised in the ICF framework, human functioning consists of complex, dynamic relationships between biopsychosocial aspects. No one research method is applicable to study all these relationships. Diﬀerent methods are required to answer clinical questions

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concerning the “measurable” aspects of phenomena relevant for practice, for instance the effect of an intervention, and the “meaning” of these phenomena, for instance the meaning children and their families attach to improving manual ability. A high-quality study, be it quantitative or qualitative, provides high level evidence when its findings are based on robust methodology that minimises bias (Howick et al., 2011). This is reflected in the revised evidence hierarchies developed by the Centre for Evidence Based Medicine (CEBM) by the University of Oxford (Howick et al., 2011).

2.6. The randomised controlled trial

Traditionally, the randomised control trial (RCT) has been referred to as “the gold standard”

method, and it has subsequently been placed on top of the evidence hierarchy for quantitive research. The RCT has gained this position because of its inherent desire to minimise bias through randomisation and strive for standardisation of the variables of interest. The RCT is useful for testing an intervention in a rigorous way at group-level; as a result, it provides the mean effect for the group and how much variation or dispersion from the average exists (the standard deviation). RCTs are appropriate only in certain phases of the development and testing of interventions, at a point when there exists a need to demonstrate that a proposed new intervention has an effect of a size that is clinically meaningful in a certain patient group (Dijkers et al., 2012, p. S168; Whyte & Barrett, 2012). When the findings from the RCTs indicate a large variation in the patients’ response to the intervention, it would be important to further study for example which individuals respond best to the intervention, or to more closely study what is the particular mechanism of action of the intervention. Depending of the identified questions, a type of experimental design, where one of the independent variable is

manipulated at a time, may provide with the best design to gain the needed evidence for applicability of the intervention for a speciﬁc population.

2.7. Systematic review & meta-analysis in EBP

With a huge growth in research publications (e.g. Sakzewski et al., 2014, p. e186), it is impossible for clinicians to keep up to date with the most current ﬁndings in primary

research. Systematic reviews provide a means to gather and critically synthesise the ﬁndings

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from primary research, and to understand the results of any single primary trial in the context of all other trials (Higgins & Green, 2011, p. 1.2.2.; Polit & Beck, 2012, p. 665; (Borenstein, Hedges, Higgins, & Rothstein, 2009, p. 9). Systematic reviews have several focus areas. On the one side, they explore the relationship between the independent variable (the variable that is manipulated, i.e. intervention) and the dependent variable (the variable that is aﬀected by the independent variable, i.e. outcome). On the other side, important knowledge is gained from reviewing the methodological quality of the primary research, and the conclusions drawn from the studies (Higgins & Green, 2011, p.2.3.1; Cooper et al., 2009, pp. 5-6). Evaluation of the quality of conduct and risk of bias in the primary research enables assessment of the validity of the primary studies, i.e. the extent to which the design and conduct of the studies likely to prevent systematic errors (Higgins & Green, 2011, p. 79). The congregated ﬁndings give important indications for future research in the focus area.

A variety of measurement instruments based on different scoring systems are typically used in the primary studies, making it challenging for clinicians to draw conclusions about the meaningfulness of the outcomes for practice. Some systematic reviews are based on a narrative analyses of the findings in the primary research, and use the p-values to assess the important of the findings in the primary research. However, the p-value is only indicative of the strength of the evidence for that the null hypothesis is false (Polit & Beck, 2012, p. 477).

The p-value can thus only indicate that the difference between the groups or the relationship between the variables is not zero, it does not give any indication about the size of the effect (Higgins & Green, 2011, p. 12). When some studies are statistically significant and others are not, the results are often interpreted as conflicting (Higgins & Green, 2011, p.12). The p-value is driven by a sample size, and therefore, as Higgins & Greene (2011, p.12) argue, reporting the p-value has no clinical importance, and basing practical conclusions on p-values is to miss the point of a review.

As an alternative, calculating effect sizes for the findings provides a way to convert the intervention outcomes measured with different instruments into a standardised value that describes the difference between the combined means of the intervention and the control groups divided by the means of the pooled standard deviation (Acton, 2001, p. 540). The effect size thus allows for comparison of the intervention effects on a standardised scale.

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A common problem in research on children with CP is the small number of available participants (Kunz, Autti-Ramo, Anttila, Malmivaara, & Makela, 2006, p. 1239). Studies with small sample sizes are susceptible to random errors and overestimation of the real eﬀect size (Acton, 2001, p. 540). A meta-analysis may therefore be useful in overcoming this problem as it allows for incorporation of the samples in the included primary trials into one large sample.

Compared to primary studies with small sample sizes, meta-analyses are able to produce a more reliable and accurate conclusion of the direction, size, consistency and variance of the eﬀect of the interventions across the primary studies (Polit & Beck, 2012, p. 654; Higgins &

Green, 2011, p. 9; Field, 2009, p. 790). It provides a framework for evaluating the outcome data from a series of studies as a whole, rather than looking at each in isolation (Borenstein et al., 2009, p. xxiv). However, conducting a meta-analysis is not always appropriate. A common critique states that meta-analysis mixes apples and oranges (Dijkers, Murphy, et al., 2012, p.

S170). However, as Polit & Beck (2012, p. 655) argue, meta-analysis should not be about

“fruit”, e.g. apples, but rather about a specific type of fruit, e.g. Granny Smith apples; a synthesis of various treatments offers little practical value (Acton, 2001, p. 542). When the heterogeneity of the primary studies is assessed to be significant, the method of choice is a systematic review with an in-depth narrative analysis of why the results are conflicting (Borenstein et al., 2009).

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3. Methods

An explorative systematic review of randomised controlled trials (RCTs) was performed using the American Academy of Cerebral Palsy and Developmental Medicine (AACPDM)

methodology (Darrah et al., 2008). The PRISMA (Preferred Reporting Items for Systematic reviews and Meta-Analyses statement) (Liberati et al., 2009) and the Cochrane Handbook for Systematic Reviews of Interventions (Higgins & Green, 2011) were used ensure quality of the review process.

Search strategy

A systematic, computer-aided literature search was performed in November 2013 and updated in April 2014. Following the AACPDM methodology, only original articles written in English and published in peer-reviewed scientiﬁc journals were included. The following databases were searched: Cumulative Index to Nursing and Allied Health Literature (CINAHL) Plus (up to week 18, 2014), the Cochrane Library (up to issue 5 of 12, May 2014), EMBASE (1980 to 2014 Week 18, 2014), Ovid MEDLINE (1946 to April Week 4, 2014), Physiotherapy Evidence Database (PEDro) (database updated 4 November 2013), PsycINFO (1806 to April Week 5, 2014), PubMed (up to week 18, 2014), The reference lists of the trials meeting the inclusion criteria were reviewed, and any missed relevant articles were added.

The search strategy was developed by MT and reviewed by Torstein Låg (PhD), the principle librarian at the Psychology and Law library at the University of Tromsø. The ‘intervention’ and the ‘comparison’ components of the search strategy were collapsed to increase the yield of possibly relevant for further manual examination. The strategy was adapted for other databases when necessary, and provided in supplementary material I. In Ovid MEDLINE the following search strategy was used:

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1. Cerebral Palsy/

2. cerebral pals*.mp.

3. Hemiplegia/

4. hemipleg*.mp.

5. 1 OR 2 OR 3 OR 4

6. (constraint adj3 therap*).mp.

7. CIMT.mp.

8. CI therap*.mp.

9. unimanual.mp.

10. Restraint, Physical/

11. 6 OR 7 OR 8 OR 9 OR 10 12. HABIT.mp.

13. BIT.mp.

14. bimanual.mp.

15. 12 OR 13 OR 14 16. 5 AND (11 OR 15)

Criteria for considering studies for this review

The inclusion and exclusion criteria applied in this systematic review is speciﬁed in table 1 with additional reasoning provided below.

Types of studies

Due to limitations in the methodology of this review, qualitative studies were excluded.

Nevertheless, following the AACPDM methodology, relevant qualitative studies were identiﬁed and the references provided (supplementary material II) as these studies supply relevant information and may be interesting for the reader.

Types of participants

Studies including children who had received Botox injections within six months prior to entering the trial were excluded. Although Botox injections alone have not been found to improve UE ability in children with unilateral CP, high-level evidence exists in that

occupational therapy combined with Botox injections has a large eﬀect in improving

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Table 1. Characteristics of the included and excluded trials.

Inclusion criteria Exclusion criteria

Types of studies

- Studies using randomised controlled group research designs

- Studies published in English in peer- reviewed scientiﬁc journals

- Systematic & non-systematic reviews - Expert opinions, case studies, conference

abstracts

- Qualitative studies - Unpublished studies

Types of participants

- Children with ‘spastic unilateral CP (hemiplegia)’ or ‘congenital hemiplegia’ aged 17 years and below

- Other subtypes of CP

- Progressive conditions, spinal diseases, traumatic brain injury

- Children who received Botox injections in the UE within 6 months prior to study entry - Children who had previously undergone CIMT

or BIT

Types of interventions

- One group receiving CIMT and one group receiving BIT within the same trial - Any type of constraint used in the CIMT

group

- Interventions conducted in all environments

- Combinations of CIMT and BIT within one group (hybrid models)

- CIMT or BIT amended with computer-based training / gaming

Types of outcome measures &

measurement instruments

- Bimanual UE function* measured with: AHA, ABILHAND-Kids

- Unimanual UE function* measured with:

MUUL, QUEST

- Participation in everyday activities*: PEDI, CAPE

- Goal achievement**: COPM

- Quality of life**: CP QOL-Child, CPCHILD

Abbreviations: AHA = Assisting Hand Assessment; MUUL = Melbourne Assessment of Unilateral Upper Limb function;

QUEST = Quality of Upper Extremity Skills Test; CAPE = The Children's Assessment of Participation and Enjoyment; COPM = Canadian Occupational Performance Measure; GAS = Goal Attainment Scale; QoL = quality of life; * = primary outcome of interest; ** secondary outcome of interest.

individualised outcomes, and a modest effect in improving quality of movement of the affected UE (Sakzewski et al., 2014, pp. e192-193) (B. J. Hoare et al., 2010, p. 2010). Studies including children who had previously undergone CIMT were excluded because children who participated in two courses of CIMT with 12 months between the interventions were found to maintain improvements from the first course of the intervention, and make further gains after the second course (Charles & Gordon, 2007).

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Types of interventions

As the terms ‘modiﬁed’ CIMT and CIMT have been used inconsistently in paediatric literature, studies using either term were included. Only the term CIMT is used in this review. No

restrictions in terms of the constraint used were applied as it has not been established that one type of constraint is more eﬀective than another (Eliasson et al., 2013, p. 131).

Interventions provided in all types of environment were included as it has not been established that one environment provides a more eﬀective context for the interventions than another (Eliasson et al., 2013, p. 132).

Types of outcome measures

The primary outcomes of interest were bimanual UE function, unimanual UE function and participation in everyday activities. The secondary outcomes of interest were achievement of individual goals and quality of life. Potential measurement instruments with adequate

psychometric properties were identiﬁed from recent systematic reviews. When the primary studies meeting the inclusion criteria employed measurement instruments that were initially overlooked, these instruments were further evaluated and included in the list of

measurement instruments when they were assessed to have adequate psychometric properties. Existing literature was used to identify the ICF-CY domains assessed on each measurement. Reports based on the standardised ICF linking rules (Cieza et al., 2005) were preferred. The identiﬁed measurement instruments are described below with table 2 providing details of psychometric properties and the ICF domains of each instrument.

Assisting Hand Assessment (AHA). The AHA is a condition-speciﬁc instrument that assesses spontaneous use of the aﬀected UE in bimanual activities (Hoare, Imms, Randall, & Carey, 2011; Krumlinde-Sundholm et al., 2007, p. 259). It is validated for children with unilateral CP or obstetric brachial plexus palsy aged 18 months to 12 years, and has excellent psychometric properties (Gilmore, Sakzewski, et al., 2010; Greaves, Imms, Dodd, & Krumlinde-Sundholm, 2010, p. 420; Klingels et al., 2010, pp. 892-895; Krumlinde-Sundholm, 2012, p. 808). The test development process is described in detail in Krumlinde-Sundholm et al. (2007) and Holmefur et al. (2007).

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The AHA assesses six domains of bimanual ability including general use, arm use, grasp and release, ﬁne motor adjustments, coordination and pace of performance. It is the only

condition-specific UE instrument where most items assess concepts in the ICF-CY activity domain, or a combination of activity and body function [see Hoare et al. (2011, p. 993)] for a detailed analysis of the ICF-CY domains. The test is criterion-referenced with each item being scored on a four-point scale (1 = does not do; 2 = ineffective; 3 = somewhat effective; 4 = effective) (Krumlinde-Sundholm et al., 2007, pp. 259-260). The scores may be presented on a logic-scale or on a logic based 0-100 AHA-unit scale (Krumlinde-Sundholm, 2012).

Melbourne Assessment of Unilateral Upper Limb Function (MUUL). The MUUL is a

condition-speciﬁc instrument that evaluates the quality of UE function in unimanual activities.

It is validated for children with CP and other neurological impairments aged 18 months to 15 years, and has excellent psychometric properties (Gilmore, Sakzewski, et al., 2010, p. 20;

Klingels et al., 2010, p. 895; Wagner & Davids, 2012). Four domains of upper extremity use are assessed including range of movement, accuracy (of reach and release), ﬂuency of UE

movement and dexterity (of grasp) (Hoare et al., 2011, p. 994). Hoare et al. (2011, pp. 990-991) provide a detailed description of the ICF-CY domains. The MUUL is criterion-referenced with each item being scored on a 3-, 4- or 5- point ordinal scale. The total test score is converted to a percentage score with a higher the percentage implying better UE function (Wagner &

Davids, 2012, pp. 1267-1268).

Quality of Upper Extremity Skills Test (QUEST). The QUEST is a non-condition speciﬁc instrument based on neuro-developmental theories that evaluates the quality of UE function in unimanual activities. It is validated for children aged 18 months to 8 years with a

neuromotor dysfunction involving spasticity, including but not limited to CP (Gilmore et al., 2010a, p. 16; Wagner & Davis, 2012; p. 1269). Although the QUEST presents with initial reliability, the administration, and scoring of some items remains unclear, and the reliability studies are based on only two raters (Gilmore et al., 2010a, p. 20).

The QUEST evaluates four domains including dissociated movements, grasp, protective extension, and weight bearing (Hoare et al., 2011, p. 992). Hoare et al. (2011, p. 992) provide a detailed description of the ICF-CY domains. The QUEST is criterion-referenced (Klingels et al.,

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2012) with each movement and task being assessed as ‘‘yes’’ (2 points), ‘‘no’‘ (1 point) or “not tested” (1 point) (DeMatteo, 1992, p. 14). Each UE is assessed and evaluated separately, and the total raw score is converted to a percentage score with a higher score representing better quality of UE movement (Wagner & Davids, 2012, p. 1269).

The Jebsen–Taylor Test of Hand Function (JTTHF). The JTTHF is a non-diagnosis speciﬁc instrument that evaluates unimanual hand use and movement eﬃciency in everyday activities (Sakzewski et al., 2011a, p. 666; Wagner & Davids, 2012, p. 1261). The test was originally

developed and validated for the adult population and for typically developing children (Boyd et al., 2013, p. 12). Its validity and reliability in children with unilateral CP has not been

established (Sakzewski et al., 2011a, p. 666). The JTTHF consists of a seven subtests including writing, ﬂipping index cards, object placement, simulated eating, stacking checkers, and manipulating empty and full cans (Gordon et al., 2011, p. 694). A detailed analysis of the ICF- CY domains has not been conducted. The child is instructed to perform the tasks as rapidly and as accurately as possible, with the total score being the sum the times in each subtest (Islam et al., 2013, p. 4). A lower score indicates better function. The test has been modiﬁed for children with unilateral spastic CP in that the writing task has been removed and the time to complete each task was reduced to 2 minutes from 3 minutes to avoid frustration (Klingels et al., 2012, p. 892).

The Besta Scale. The Besta scale is condition-speciﬁc instrument consisting of three domains of UE function including grasp, bimanual use and activities of daily living that together

compose a global score (Fedrizzi et al., 2013, p. 164). It has good interrater reliability (Fedrizzi et al., 2013, p. 164); other psychometric properties have been poorly evaluated or no

information could be found. No analyses of the ICF-CY domains have been conducted. The items are assessed on four-point discrete, ordinal level scales. The quality of the grip is scored as follows: 0 = inability to grip cube; 1 = grasping or whole-hand grip; 2 = radial or three-ﬁnger grip; 3 = pincer grip. Spontaneous hand use and activities of daily living are scored based on the variability and stereotypy of movement patterns as follows: 0 = no use of impaired limb; 1

= use of impaired limb in a stereotyped pattern (wrist support); 2 = cooperation of the

impaired hand in manipulation by holding with a restricted number of stereotyped patterns; 3

= cooperation of the impaired hand with holding and manipulation, using a varied repertoire of patterns (Facchin et al., 2009, p. 224).